Zebrafish: A Model Organism for Studying Enteric Nervous System Development and Disease
Open Access
- 21 January 2021
- journal article
- review article
- Published by Frontiers Media SA in Frontiers in Cell and Developmental Biology
Abstract
The Enteric Nervous System (ENS) is a large network of enteric neurons and glia that regulates various processes in the gastrointestinal tract including motility, local blood flow, mucosal transport and secretion. The ENS is derived from stem cells coming from the neural crest that migrate into and along the primitive gut. Defects in ENS establishment cause enteric neuropathies, including Hirschsprung disease (HSCR), which is characterized by an absence of enteric neural crest cells in the distal part of the colon. In this review, we discuss the use of zebrafish as a model organism to study the development of the ENS. The accessibility of the rapidly developing gut in zebrafish embryos and larvae, enables in vivo visualization of ENS development, peristalsis and gut transit. These properties make the zebrafish a highly suitable model to bring new insights into ENS development, as well as in HSCR pathogenesis. Zebrafish have already proven fruitful in studying ENS functionality and in the validation of novel HSCR risk genes. With the rapid advancements in gene editing techniques and their unique properties, research using zebrafish as a disease model, will further increase our understanding on the genetics underlying HSCR, as well as possible treatment options for this disease.Funding Information
- Stichting Vrienden van het Sophia
This publication has 118 references indexed in Scilit:
- Hirschsprung-like disease is exacerbated by reduced de novo GMP synthesisJCI Insight, 2013
- RNA-Guided Human Genome Engineering via Cas9Science, 2013
- Multiplex Genome Engineering Using CRISPR/Cas SystemsScience, 2013
- A Programmable Dual-RNA–Guided DNA Endonuclease in Adaptive Bacterial ImmunityScience, 2012
- Efficient design and assembly of custom TALEN and other TAL effector-based constructs for DNA targetingNucleic Acids Research, 2011
- Involvement of SOX10 in the pathogenesis of Hirschsprung disease: report of a truncating mutation in an isolated patientJournal of Molecular Medicine, 2010
- The Tol2-mediated Gal4-UAS method for gene and enhancer trapping in zebrafishMethods, 2009
- Endoderm-derived Sonic hedgehog and mesoderm Hand2 expression are required for enteric nervous system development in zebrafishDevelopmental Biology, 2008
- Sequential actions of Pax3 and Pax7 drive xanthophore development in zebrafish neural crestDevelopmental Biology, 2008
- Stages of embryonic development of the zebrafishDevelopmental Dynamics, 1995